ejbps, 2017, Volume 4, Issue 11, 91-95. Case Report SJIF Impact Factor 4.382
PHARYNGEAL HEMORRHAGE: A CATASTROPHIC POST-OPERATIVE EVENT
*1Giulio Rizzoli MD FETCS and 2Ivo Tiberio MD
1Division of Cardiac Surgery, University Hospital, Padova, Italy.
2Anesthesia and Intensive Care, Sant’Antonio Hospital, Padova, Italy.
*Corresponding Author: Giulio Rizzoli MD FETCS
Division of Cardiac Surgery, University Hospital, Padova, Italy.
Article Received on 09/09/2017 Article Revised on 30/09/2017 Article Accepted on 21/10/2017
Hematoma of pharyngeal muscles is an unknown and potentially fatal cause of airway obstruction. We report a case of a 71 year old woman who received a long lasting combined, carotid + CABG, operation and developed severe dispnoea and stridor post extubation. Underwent an unrecognized esophageal intubation and died of brain edema. Autopsy showed the presence of massive hematoma of the pharyngeal muscles, possibly related to minor pharyngoesophageal trauma in a double anti-aggregated patient.
KEYWORDS: esophageal intubation, pharyngeal hematoma.
71-year-old female patient with history of smoking, familiarity for ischemia and atrial fibrillation dating several years, dyslipidemic and hypertensive, assuming a major therapy with anti-aggregating agents (aspirin and clopidogrel), beta blockers, ACE inhibitors, protonic pump inhibitors, statins and nitrates. She complained of inducible ischemia after acute lower-infarction, complicated from ventricular fibrillation and DC shock in October 2007. Coronarography showed double vessel disease and on that occasion, a rescue angioplasty with stent deployment within the right coronary (RC) was done. The following month she underwent new deployment of two medicated stents (Cypher) on the anterior descending (AD) coronary artery.
Nonetheless on may 2008 there was evidence of positive ErgometricTest with a low ischemic threshold. A repeated coronarography evidenced intrastent restenosis (75%) of RC artery, therefore a repeated angioplasty was made with intrastent insertion of a new medicated stent. Furthermore the AD showed 70% stenosis upstream of the previous stent and critical stenosis of the 1st diagonal branch of good caliber plus 40% stenosis of the circumflex (CX) branch.
The ejection fraction (EF) was 55% with akinesis of lower basal septum and hypokinesis of the median and basal inferior wall and of the lateral inferior wall. There was an aneurysm of the interatrial septum and the ECG showed episodes of paroxismal atrial fibrillation. The carotid vessel were affected from 70% critical stenosis on the right side and a moderate stenosis (50%) on the left. Risk of surgical mortality was estimated 4.25% according to Euroscore. STS score estimated a 9.6% combined probability of morbidity or mortality and 1.2% probability of stroke.
On July 2008 we proceeded to right carotid endoarterectomy (CEA) with polytetrafluoroethylene (PTFE) patch, followed by median sternotomy and take down of both mammary arteries. Thereafter extracorporeal circulation (ECC) was initiated and the heart arrested with antegrade and retrograde blood cardioplegia. We then proceeded to left atriotomy and radio frequency ablation of the orifices of the lung veins, right atriotomy and plication of the aneurysmal interatrial septum, double aorto-coronary bypass (CABG) with right mammary artery to the right coronary and left mammary artery to the anterior descending. ECC weaning and heart recovery was monitored using transesophageal echocardiography.Mediastinal and pleural drainage were placed and the neck was sutured over a vacuum drainage at the completion of the eight hour procedure. The patient was transferred to the intensive care unit (ICU) at 5 pm.
The extubation maneuver was postponed for hypertension and shiver, treated with Venitrin infusion and sedation. The neck drainage (200 cc) as well as the chest drainage were trivial.There was a mild neck edema in the sub-mandibular and lateral neck region and some bulging of the sternocleid muscle but an iced bag was thought not necessary. Chest X ray was clear and the trachea was straight. After recovery of temperature and consciousness she was extubated at 11 pm. She was noted to be alert and responding to questions with a normal voice, the serving nurse reported that the patient was quiet, frequently asking for water sips and that she had gradually dozed. Thirty minutes later patient’s conditions rapidly deteriorated because of a severe respiratory distress with stertorous breathing. The eyes were wide open, rotated upward and with dilated pupils. The patient was no longer contactable and there was a spastic extension of the left arm which was internally rotated, the whole suggesting a possible brain damage. Arterial pressure was 240 mmHg.
The most frequent complications of carotid TEA, namely an Ictus, was suspected, a diagnosis further supported by the additional risk factors for clot embolism, id est the surgical radioablation of the orifices of pulmonry veins in the left atrium and the plication of the aneurysmal interatrial septum. An alternative hypothesis was cerebral edema, secondary to high blood pressure that the patient had suffered from postoperatively and she was suffering again at the time.It was therefore necessary to reintubate the patient, a maneuver that was performed from the resident on call with a rapid sequence intubation using ketamine, fentanil and rocuronium bromide, nonetheless, within the matter of a minute a sudden drop of blood pressure with cardiopulmonary arrest followed.
The immediate hypothesis was of an intubation failure, possibly esophageal. The external heart massage was immediately initiated and the anesthesiologist was requested to verify the position of the tube and eventually replace it: He was definitely confident in the appropriate tube positioning and refused to proceed. Since resuscitating maneuvers had no success and time was running, we again urged the anesthesia resident to carefully verify the presence of lung ventilation or the presence of air in the stomach but again he felt that there was no evidence of malposition, forcing us to search for different solutions. After several minutes the senior anesthesiologist on duty arrived, he also verified the absence of clinical signs of malposition but, being aware of the in house presence of a videolaryngoscope (Glidescope), sent for it . The device documented an esophageal intubation. Because of the severely restricted airway a new smaller tube was inserted, the lung was finally ventilated, with recovery of the sinus rhythm after defibrillation. At the end of the procedures we noticed that the neck had considerably swollen. There was a significant drop of the emoglobin from 10.3gm to 8.2gm. The anoxyc lapse was unfortunately prohibitive and she died a few days later because of severe brain edema.
The autopsy report: There was hemorrhagic infiltration of ipsilateral sternocleid muscle and a clot, due to the use of several sealants on the carotid suture, that was marginally imprinting the giugular vein. There was a diffuse tissue edema but lack of blood collections or traces of bleeding in the neck and in the loose compartments between pharyngeal and columnar albuginea. There was no trace of any surgical penetration of the tick carotid fascia.The carotid suture was water proof.On the contrary the pathologist observed an intramuscular hemorrhage of the middle and lower muscle of the pharynx resulting in a massive muscular infarction that “involves the entire thickness of the muscle”, a finding that impressed and surprised him, who collected and preserved it.
The unrecognized esophageal intubation resulted in demise of the patient. There are several lessons to be learned from this case. Nowadays It is rather uncommon to intubate the esophagus because a large experience was accumulated in the recent years and clinicians may rely on many new devices to avoid or to recover from this mistake. In the early years of the century the incidence of this potentially tragic error was estimated as high as 2.7%. There was evidence that this mistake can equally affect unexperienced and expert professionals, but the expert one are probably better prepared to deal with it.
At the time the anestesiology service was staffed with residents. A II° year resident was on duty that night, supported from a senior anesthesiologist on call. Video laryngoscope, that was largely employed in the teaching units of the US since 2001 and had been shown to offer a prominent advantage in the case of difficult intubation, was unsupported. The department was just starting to evaluate the utility of the “Glidescope”, but this device was not yet available in the intensive care unit (ICU).ICU lacked capnography modules,which have 100% sensitivity and specificity for detection of tube misplacement, or a capnometry probe equally sensible but less specific (88%).
The preoperative anesthesiology visit categorized our patient as Mallampati class 2  therefore an intubation within a resident expertise was expected.The control of tube position within ICU was routinely done with a portable anterior-posterior chest X-Ray, which can exclude a carotid hematoma when trachea and esophagus are properly aligned. The resident was confident to have passed the tube within the trachea under direct vision but this verification, although logically sound, may be misleading because he was deceived from a disrupted anatomy. Furthermore he was sure to hear ventilation sounds, which can nonetheless be easily mimicked from air passing trough the esophagus, and was not able to identify the flow of air in the stomach. Andersen and coworkers  demonstrated that all this clinical methods failed to identify esophageal intubation in at least 1 of 40 cases and should never be relied upon as the sole method of verification.
Anatomic disruption: Diffuse edema of the airway and the pharynx is a common finding after carotid surgery. In 1977 a TAC study of 38 patients operated on unilateral carotid endarterectomy at Toronto Hospital showed that edema of the neck is inevitable after carotid surgery even without hematoma. All had significant airway and neck edema that in most cases extended to the posterior pharyngeal space. None of these patients needed to be re-intubated. In this setting the occurrence of a blood hematoma is the classic “straw that breaks the camel back”. Carmichel underlines that CT scan showed soft tissue swelling in all 19 patients but five patients with clinical evidence of airway obstruction and an hematoma were intubated postoperatively. He concluded "In summary, after carotid endarterectomy, all patients had significant soft tissue swelling of the neck assessed by CT scan, the presence of hematoma may increase the degree of airway obstruction requiring intubation of the trachea to maintain a patent airway. Supraglottic pharyngeal edema seen on CT scan and during laryngoscopy plus tracheal deviation can make intubation of these patients very difficult." We were obviously aware that carotid surgery may be complicated in 2-3% of cases from bleeding in the neck causing tracheal compression due to deformation and counter-lateral deviation of the trachea on the chest X-Ray  which require urgent surgical decompression, but this was not our case.
Our Case: the most interesting lesson we acquired from this case was the cause of the anatomic disruption. On December 1988 Wells  published an article commenting 6 cases originally described in 1986 in his own hospital from O’Sullivan  and characterized from an extreme difficulty in airway management, due to a heavy edema of the oropharynx, not justified by the amount of neck hematoma, which was completely absent in 3 cases. In his paper he writes “Potentially life threatening massive supraglottic swelling is occasionally seen in patients presenting with neck swelling following carotid endarterectomy”. This is considered to be a distinct clinical entity and a theoretical mechanism based on a review of the relevant anatomy and physiology was postulated and thought to be presumably due to obstruction of the prevertebral lymphatic chain.He named this syndrome Massive Supraglottic Swelling (MSS). Four of these patients were hypoxic during reintubation attempts and developed bradicardia and asystole, one sustained an irreversible brain damage, all these patient shared in common the treatment with anti-platelet agents.
Retro esophageal hemorrhage is an extremely rare event. In 1999 Senthuran described the case of a 68y/o woman who had a large cervicomediastinal hematoma that caused life-threatening airway obstruction.and enumerates a range of conditions that could cause it, including cervical trauma, anticoagulation, bleeding diathesis, complication of internal jugular vein cannulation, arteriography, carotid sinus massage and spontaneously, with the commonest cause due to cervical trauma[14.
]Post-Mortem of our patient has shown massive infarction of the pharyngeal muscles causing obstruction of the prevertebral lymphatic chain with the build up of a severe neck edema and MSS. Pharyngeal hematoma that heavily infarcted the midlle and inferior pharyngeal constrictor muscle, is extremely rare and we are not aware of a single case related to carotid surgery. Furthermore at difference with retrofaringeal hemorrhage, an intramuscular hematoma is not amenable to surgical drainage.This pathology is known from neurosurgeon operating on the column or stellate ganglion. Kazuo Higa  made a meta-analysis of 27 cases (only 4 published in English) of pharyngeal hematoma after blocking the stellate ganglion, concluding that this complication can not be predicted by signs or initial symptoms. Old people who take blood thinners or antiplatelet medications (aspirin or clopidogrel) are prone to hematoma formation. Our patient was on aspirin until the preoperative day and on clopidogrel until 3 preoperative days and was heparinized during several hours of a long lasting surgery. Autopsy showed a site of hemorrhage within the sternocleid muscle, possibly justified by the use of surgical retractors, and of the medium and lower pharyngeal constrictors without any apparent reason.
Muscular tissue is the most vascularized body tissue, therefore his bloody swelling is extremely rapid and among people assuming anticoagulant medications the occurrence of hematoma of the ileopsoas or rectus abdominis muscle is not rare. Miller found in the english literature 39 cases of spontaneous non traumatic hemorrhage of the pharyngeal region with no other cause. The single case of pharyngeal hematoma reported in the cardiothoracic literature was due to a trivial blunt trauma to the left shoulder in a anticoagulated patient. Our patient underwent several traumatic manouvers affecting the pharynx, including intubation, extubation, nasogastric tube and transesophageal ecocardiographic probe. The hypothesis of an ongoing undetectable hemorrhagic episode is supported from a 2 gm.drop of hemoglobin across the episode, despite 1 unit transfusion. In a propensity score analysis Clopidogrel discontinuation within 5 days from surgery was independently associated with transfusion requirement (P=0.001). The hematoma it is neither palpable nor visible from the outside and the Capp’s triad, the constellation of clinical and imaging finding in patients with spontaneous retropharyngeal hematomas, id est evidence of dyspnoea or stridor, subcutaneous bruising over the neck and anterior chest, anterior displacement of the trachea, was restricted to the first sign because of the thoracic surgical wound and the lack of a lateral x-ray in the routine monitoring of these patients.
Prevention and therapy: Could this complication be prevented? The vascular operation could have been.done on a separate session.Unfortunately postoperative stroke remains one of the most devastating complications of CABG, while cardiac events represent the most dangerous not-neurologic complication after CEA. Therefore our choice for a combined operation which in our hands  has comparable risk and reduces the expense of public health service.
This patient had received combined Clopidogrel and Aspirin therapy and this combination is associated with an increased risk of postoperative bleeding. While aspirin alone is well tolerated, data on the role of Clopidogrel on perioperative bleeding risk are uncertain. An increased hemorrhagic risk emerged in patients undergoing CABG while on Clopidogrel therapy, which was reduced by stopping the drug at least five days prior to intervention. Scott D Wait observes that in patients taking clopidogrel within 5 days of CEA intervention: "we noted non-operative (ie no urgent extemporaneous demanding) neck swelling more frequently in the clopidogrel group 28.6 vs. 12.9%, p = 0.030)". A metanalysis of relevant cardiac surgery studies published between 2001 and 2010 consistently showed a significantly higher rate of blood transfusion (OR: 1.82;95% CI: 1.40-2.37; p<0.00001) and reoperation (OR: 2.15; 95% CI: 1.38-3.34; p<0.00001).
Could the dismal course of the patient be avoided with emergent cricothyrodotomy? This is an invasive recovery manouver that should be in the core skill of every anesthesiologist, provided he realizes that he can’t nor ventilate nor intubate,Nonetheless, with hindsight, it would have been a risky maneuver because the patients was receiving an external massage, had a swollen neck and her backward tracheal wall was compressed and pushed forward from behind and therefore at risk of knife injury, furthermore a dedicated surgical set was not available. In the diagnostic uncertainty the safer,videolaryngoscopy guided, tube replacement was chosen.
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